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Patient-specific naturally gene-reverted induced pluripotent stem cells in recessive dystrophic epidermolysis bullosa.
[dystrophic epidermolysis bullosa]
Spontaneous
reversion
of
disease-causing
mutations
has
been
observed
in
some
genetic
disorders
.
In
our
clinical
observations
of
severe
generalized
recessive
dystrophic
epidermolysis
bullosa
(
RDEB
)
,
a
currently
incurable
blistering
genodermatosis
caused
by
loss
-of-function
mutations
in
COL
7
A
1
that
results
in
a
deficit
of
type
VII
collagen
(
C
7
)
,
we
have
observed
patches
of
healthy-appearing
skin
on
some
individuals
.
When
biopsied
,
this
skin
revealed
somatic
mosaicism
resulting
in
the
self-correction
of
C
7
deficiency
.
We
believe
this
source
of
cells
could
represent
an
opportunity
for
translational
'
natural
'
gene
therapy
.
We
show
that
revertant
RDEB
keratinocytes
expressing
functional
C
7
can
be
reprogrammed
into
induced
pluripotent
stem
cells
(
iPSCs
)
and
that
self-corrected
RDEB
iPSCs
can
be
induced
to
differentiate
into
either
epidermal
or
hematopoietic
cell
populations
.
Our
results
give
proof-of-principle
that
an
inexhaustible
supply
of
functional
patient-
specific
revertant
cells
can
be
obtained--potentially
relevant
to
local
wound
therapy
and
systemic
hematopoietic
cell
transplantation
.
This
technology
may
also
avoid
some
of
the
major
limitations
of
other
cell
therapy
strategies
,
e
.
g
.
,
immune
rejection
and
insertional
mutagenesis
,
which
are
associated
with
viral-
and
nonviral-mediated
gene
therapy
.
We
believe
this
approach
should
be
the
starting
point
for
autologous
cellular
therapies
using
'
natural
'
gene
therapy
in
RDEB
and
other
diseases
.
Diseases
Validation
Diseases presenting
"induced pluripotent stem cells (ipscs"
symptom
adrenomyeloneuropathy
dystrophic epidermolysis bullosa
severe combined immunodeficiency
werner syndrome
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